Post-traumatic growth in family members of childhood cancer survivors—an updated systematic review : Journal of Psychosocial Oncology Research and Practice

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Post-traumatic growth in family members of childhood cancer survivors—an updated systematic review

Halldorsdottir, Berglinda; Michel, Giselaa; Baenziger, Juliaa,b,c,d,*

Author Information
Journal of Psychosocial Oncology Research and Practice 4(4):, October-December 2022. | DOI: 10.1097/OR9.0000000000000087
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Abstract

1. Background

Childhood cancer can be a traumatizing experience for all family members.[1] For a long time, the focus of research on the experience and impact of childhood cancer in the family was on negative outcomes such as elevated post-traumatic stress symptoms (PTSS) in parents of childhood cancer survivors (CCS).[2] Over the last 2 decades, there has been growing attention on positive psychological consequences that can co-occur with the negatives after a traumatic event.[3]

Post-traumatic growth (PTG) was defined by Tedeschi and Calhoun (1996, 2004)[4,5] as the perception of positive changes after a stressful event. PTG involves cognitive processes such as meaning-making after a traumatic event, which may include benefit finding and personal development beyond the original level of psychological functioning.[4,5] More specifically, the PTG framework includes 5 domains: improved interpersonal relationships, recognition of new possibilities in one's life course, higher personal strength, spiritual or religious change, and appreciation of life.[4,5] The Post-traumatic Growth Inventory (PTGI) has been widely used to assess those domains of PTG and validated in numerous population and languages.[4] Another common terminology to describe perceived positive changes is benefit finding.[5] The Benefit Finding Scale (BFS) consists of 6 domains including acceptance, empathy, appreciation, family orientation, positive self-view, and reprioritization.[6]

The systematic review by Duran (2013) regarding PTG in family members of CCS showed that family members also experience higher appreciation of life, high self-knowledge, positive attitudes toward the family, a desire to pay back society, and that meaning-making of the cancer experience was important.[7] Duran found that growth was positively associated with female sex, better health, lower education, and the perception of the (grand)parenting role.[7,8] Namely, (grand)parents with higher perceived family cohesion and those who put more significance on the role, such as receiving a purpose in life and connecting with family, reported higher PTG.[7,8] Siblings of children with cancer can also experience positive changes, such as increased maturity, closer family relationships and compassion, more resilience, and improved family relationships.[7,9,10] Little is known about characteristics associated with siblings' PTG.[7]

Since the publication of Duran's review additional studies have addressed the topic of PTG in family members of CCS, and we updated the review of Duran with the following aims: We describe (1) the level and domains of PTG in caregivers (mothers, fathers, grandparents, other support persons), (2) the level and domains of PTG in siblings, and (3) characteristics associated with PTG among all family members of CCS.

2. Methods

We followed the PRISMA guidelines to transparently report the rationale, methods, and presentation of the findings of the present systematic review.[11]

2.1. Literature search

Pertinent studies were identified by systematically searching 4 electronic databases: PubMed, PsycINFO, CINAHL, and SCOPUS. The search terms were adopted from the previous review and tailored for each database to meet their specific requirements (Table 1). The references of the included studies were screened to ensure the inclusion of all relevant articles.

Table 1 - Search terms for the systematic review on post-traumatic growth in family members of CCS
Block 1:
Children and adolescents
child OR child* OR kid* OR pediatric OR paediatric* OR girl* OR boy* OR toddler* OR adolescent OR adolescen* OR preteen* OR youth* OR teen*
Block 2:
Cancer
cancer* OR neoplasms OR neoplasm* OR malign* OR tumor* OR tumour* OR oncolog* OR carcinoma OR carcinoma* OR leukemia OR leukemia* OR leukaemia* OR lymphoma OR lymphoma* OR sarcoma OR sarcom* OR blastoma* OR radiotherapy OR radiotherap* OR chemotherapy OR chemotherap*
Block 3:
Survivor (as MeSH term)
Survivor*
Block 4:
Family members
grandparents OR grandparent* OR “grand parent” OR “grand parents” OR grandmother* OR “grand mother” OR “grand mothers” OR grandfather* OR “grand father” OR “grand fathers” OR grandchild* OR “grand child” OR “grand children” OR granddaughter* OR “grand daughter” OR “grand daughters” OR grandson* OR “grand son” OR “grand sons” OR parents OR parent* OR mothers OR mother* OR fathers OR father* OR daughter* OR son* OR siblings OR sibling* OR brother* OR sister* OR spouses OR spouse* OR husband* OR wife OR wive* OR daughter* OR son* OR caregivers OR caregiver* OR carer* OR family OR famil* OR relative*
Block 5:
Post-traumatic growth
posttraumatic growth OR postraumatic growth* OR PTG OR stress-related growth OR stress-related growt* OR benefit finding OR benefit finding* OR finding benefit* OR find benefit OR thriving OR thriv* OR positive transformation OR positive transformati* OR positive psychological change OR transformational coping OR adversarial growth* OR perceived benefit* OR construing benefit* OR perception of change OR postive growth* OR positive change OR personal growth OR positive aspect* OR positive emotion* OR positive reinterpretation OR discovery of meaning* OR positive by-products OR positive adjustment

2.2. Study eligibility criteria

The inclusion criteria were as follows: (1) use of quantitative, qualitative, or mixed methods; (2) published in English language peer-reviewed journals (March 2010 to December 9, 2021); (3) assessing PTG in family members of CCS younger than 21 years at diagnosis; (4) for the purpose of this review, survivors were defined as having completed cancer treatment before study participation and no relapse at the time of study; and (5) studies had to state clearly whether participants were parents, grandparents, or siblings of the CCS. Studies were excluded if the above criteria were not met or the study was about bereaved family members.

2.3. Study selection

Titles and abstracts were screened independently for inclusion/exclusion by 2 authors (B.H., J.B.; Fig. 1). For the remaining studies, the full text was independently read by 2 authors (B.H., J.B.) to assess eligibility. Any disagreements raised between the authors were resolved through discussion and inclusion of a third opinion when appropriate (G.M.) (agreement B.H./J.B. 424/434 = 97.7%). Disagreements were most often related to uncertainties regarding time since diagnoses or treatment end, which required careful examination of the data presented in the articles.

F1
Figure 1.:
Inclusion flowchart for the systematic review on post-traumatic growth in family members of CCS.

2.4. Data extraction

We systematically extracted study data including authors, publication year, study origin, study design, number of participants, age of survivors at diagnosis (mean, range), age of survivors at time of study (mean, range), and time since end of treatment (mean, Table 2). We further extracted a summary of the main PTG results, including levels and domains (aims 1 and 2), and all characteristics that were reported to have been investigated for potential association with PTG outcomes (aim 3, Table 2).

Table 2 - Data extraction for the updated systematic review “Post-traumatic growth in family members of childhood cancer survivors”
Authors (publication year) Study origin Study design Number of participants Age of CSS at diagnosis: mean (range) Age of CSS at start of study: mean (range) Time since end of treatment: mean (range) Reporter of growth Measure of growth Main results (aims 1 and 2) Associations (aim 3)
Castellano-Tejedor et al (2017) [16] Spain Quantitative and cross-sectional 41 parents of CCS (8 fathers and 33 mothers) 11.76 (8–16) 17 (13–20) Semistructured interview Positive consequences: “do you consider you have obtained something good from the oncological experience you have been through? Nine (22%) parents reported uniquely positive consequences resulting from the childhood cancer experience. Twenty-four (58.5%) both positive and negative, 3 (7.3%) uniquely negative, and 5 (12.2%) perceived no consequences. Demographic and clinical. No significant differences were found for parental sex and survivor's age, sex, diagnosis, sequelae, and time elapsed since diagnosis for reporting positive changes vs not.
Findler et al (2014) [17] Israel Quantitative and cross-sectional 56 grandparents of CCS; 60 grandparents of healthy children in comparison group 4–12 Self-report PTGI Grandparents of CCS reported higher PTG than grandparents of healthy children. Psychological. No association of PTG with sense of coherence, and social support (family/friend/partner) was found in grandparents of CCS; in contrast, friend and family support was associated with PTG in grandparents of healthy children.
Ford et al (2015) [18] USA Quantitative and cross-sectional 2820 siblings of retinoblastoma survivors 1 (median) (0–17) 43.3 Self-report PTGI No significant difference in any subscale of PTG domains was found between survivors of retinoblastoma to their siblings after adjusting for age at study, race/ethnicity, household income, and education. NA
Gardner et al (2015) [20] USA Quantitative and cross-sectional 83 caregivers of CCS 5.93 (0–17) 14.01 (4–27) 5.71 Self-report BFS NA Demographic. No associations found for: parental age, ethnicity, education level, religion, family composition, relationship to child, urban/rural designation, child age, sex, and ethnicity. Higher family income was associated with higher benefit finding.
Clinical. Higher benefit finding was found for caregivers of children diagnosed with leukemia and those treated with only 1 modality, ie, surgery or chemotherapy or radiation. No relapse was associated with higher benefit finding.
Psychological. Higher level of active, emotion, positive spiritual coping, social support, optimism, and lower perceived illness impact was associated with greater benefit finding after adjusting for demographic and clinical variables. Acceptance coping was not associated with benefit finding.
Demographic and clinical variables were not significantly associated with benefit finding in multivariable model including the psychological variables.
Gardner et al (2017) [19] USA Quantitative and cross-sectional 83 caregivers of CCS 5.93 (0–17) 14.01 (4–27) 5.71 Self-report BFS NA Quality of life was positively associated with benefit finding. The associations between quality of life and benefit finding were stronger for those younger, unmarried, with lower SES, living in rural (vs urban) areas, reporting higher post-traumatic stress, higher caregiving burden, using avoidant or emotion coping, and were less optimistic. This interaction was not found for active and acceptance coping and social support.
Hensler et al (2013) [34] USA Mixed methods 25 fathers of CCS 5.7
11.7
4.5 Self-report
Interview
BFS
Open-ended questions regarding benefit finding
High levels of benefit finding: mean = 4.1 of 5 (extremely).
Three themes were identified: personal/inner strength (helping to effectively deal with future life adversities), spiritual change (questioning vs deepening faith and belief systems), and relating to others (quality time, creating memories together). All fathers experienced change in 1, 48% in 2, and 16% in all 3 of these domains.
NA
Kamibeppu et al (2010) [28] Japan Quantitative and cross-sectional 72 siblings of 185 CCS and 658 survivor-matched controls 8 (0–18) 23.15 (16–40) Self-report PTGI-J (Japanese version) Total PTG and ‘relating to others’ was higher in female siblings compared with female controls. For both male and female siblings, ‘appreciation of life’ was significantly higher than in the controls. Controls were matched for residential area, age, and sex of CCS. NA
Ljungman et al (2016) [30] Sweden Qualitative 132 parents of CCS (68 mothers and 64 fathers) 0–18 Telephone interview Semistructured open-ended question “Have you had any particularly positive experience in relation to your child's cancer? Fifty-nine (92.2%) of fathers and 61 (89.7%) of mothers of CCS reported positive experiences. Of 7 categories in positive experiences, 3 regarded PTG including improved relationships, positive long-term development for the child, and personal development. NA
Molinaro et al (2018) [31] Canada Qualitative 9 primary support person of 10 CCS 10.6 (2–19) 24 (21–28) Interview Semistructured questions regarding experiences; then selected those which were positively appraised for analyses of PTG PTG was reflected in cherishing life and helping others, including experiences stronger and closer family relationships and wanting to give back to society, eg, doing charity work or mentoring other children under cancer treatment. NA
Porteous (2019) [32] New Zealand Qualitative 10 siblings of CCS 3.6 Interview Semistructured; asking about memories of the childhood cancer experience, including perception of personal change Siblings of CCS all expressed some positive aspect of their experiences of sibling cancer.
The main theme of personal growth was that siblings felt an increased maturity and independency as direct result of the childhood cancer experience. Siblings also reported identifying positives about difficult situations, new opportunities, and a desire to make most of life and relationships. A little less than half of siblings reported that the family had grown closer after the cancer experiences, even when parents separated. Changes included sibling relationship, improved communication, and appreciation of time with family.
NA
Schepers et al (2018) [21] USA Quantitative 181 mothers and 25 fathers of 206 CCS; 119 mothers, 13 fathers of healthy children 7.98 15.04 (8–21) 5.78 Self-report PTGI Parents of CCS reported higher PTG, higher attachment, and less relational frustration in the parent-child relationship than parents of healthy children. NA
Turner-Sack et al (2016) [22] Canada Quantitative and cross-sectional 30 parents and 18 siblings of 31 CCS 7.45 15.74 6.47 (2–10) Self-report PTGI Parents of CCS experienced average but higher PTG (M = 2.83 of 0 [no]–5 [high] possible) than CCS (M = 2.15). Siblings reported low PTG (M = 1.84). Demographic and clinical. There were no significance association found for age, sex, ethnicity, education, diagnosis, age at diagnosis, time since diagnosis, time since treatment completion, and length if treatment with PTG among parents or siblings of CCS.
Psychological. Active coping in CCS was positively associated with higher parental PTG. Parental distress was negatively associated with parental PTG.
Wataru et al (2019) [23] Japan Quantitative and cross- sectional 78 parents of CCS; 44 parents of children with chronic disease 10.17 Self-report PTGI-J (Japanese version) Reported PTG was not different in parents of CCS (M = 46.8) and parents of children with other chronic diseases (M = 44.1). Demographic and clinical. Parents' age, child's age, and time since diagnosis.
Psychological. For the parents of CCS (but not parents of chronically ill children), the re-examination of core beliefs was more strongly associated with PTG than deliberate rumination.
Willard et al (2016) [35] USA Mixed method 40 maternal caregivers of CCS 4.0 (0–15) 10.6 (3–17) 4.7 (1.0–13.2) Self-reported narrative questionnaire “Have there been any benefits, or positive outcomes, as a result of your experience of having a child with cancer? If so, how would you describe them? Most (n = 33, 82.5%) of maternal caregivers of CCS reported some type of benefits (1–6 benefits identified) from experience of having child with cancer, categorized into social support (n = 20, 50%) referring to stronger family unit; personal growth (n = 31, 77.5%) in the maternal caregivers including appreciation of life, inner strength, and increased faith and spirituality; and child-specific growth such as benefits perceived in their children (eg, character changes “stronger” “funnier” “braver”). Psychological. Parents with more emotional resources and positive reappraisal coping reported more benefits. Parental guild and worry, unresolved sorrow, long-term uncertainty, and accepting responsibility were associated with reporting less benefits.
Yonemoto et al (2012) [15] Japan Quantitative and cross-sectional 34 families of 34 CCS including 30 fathers and 28 mothers 12.9 (7–18) 18.2 (3.6–31.2) Self-report PTGI Mean [M] PTG was 44.9, with mothers' M = 46.4 and fathers' M = 42.8. Demographic and clinical. No significant association was found with parental age at time of the survey and at time of study, respectively, parents' sex, time after end of treatment, or state of affected limb in patients with parental PTGI.
Psychological. Increased levels of post-traumatic stress symptoms (intrusion, avoidance, hyperarousal) were significantly associated with higher PTG among parents of CCS.
Zebrack et al (2011) [24] United States Quantitative and cohort 360 siblings of 6425 CCS 8.7 (0–20) 32.3 (18–40+) Self-report Modified version of PTGI Siblings perceived lower positive impact in all PTG domains than the survivors did, adjusted for age, sex, and race, with small to moderate effect sizes (0.22–0.37). NA
—, not reported; NA, not available; QOL, quality of life; SES, social economic status.

2.5. Quality appraisal

We appraised studies for methodological quality (B.H., J.B.) using the Mixed Methods Appraisal Tool (MMAT),[12] including 4 criteria for (1) qualitative studies; (2) nonrandomized quantitative studies; and (3) mixed methods studies (Tables 3 and 4). The response for each item was either “yes,” “can't tell,” or “no.” Studies received one * for each criteria responded by “yes.”[12] Mixed methods studies scored the qualitative and quantitative components separately with the lower of the 2 taken as the overall quality score. Quality appraisal was performed independently by the 2 authors (B.H.,J.B.). Disagreements were discussed and resolved (10/108 of cases); disagreements most often related to the representativeness of the population under study, and answers were changed from “yes” to “can't tell” based on the information provided in the articles.

Table 3 - Post-traumatic growth—quality assessment of the studies (qualitative and mixed methods) using Mixed Methods Appraisal Tool
First author (year) Type of study Screening questions Qualitative
Are there clear research questions? Do the collected data allow to address the research questions? 1.1. Is the qualitative approach appropriate to answer the research question? 1.2. Are the qualitative data collection methods adequate to address the research question? 1.3. Are the findings adequately derived from the data? 1.4. Is the interpretation of results sufficiently substantiated by data? Quality
x/****
Hensler et al+ (2013) [34] Mixed methods Yes Yes Yes Yes Can't tell Yes ***
Ljungman et al (2016) [30] Qualitative Yes Yes Yes Yes Yes Yes ****
Molinaro et al (2018) [31] Qualitative Yes Yes Yes Yes Yes Yes ****
Porteous et al (2019) [32] Qualitative Yes Yes Yes Yes Yes Yes ****
Willard et al+ (2016) [35] Mixed methods Yes Yes Yes Yes Can't tell Yes ***
NOTE: For qualitative and quantitative studies: * = 1 criterion met; ** = 2 criteria met; *** = 3 criteria met; **** = 4 criteria met. For mixed methods studies, the lower of the 2 is taken as the overall quality score; + = mixed method study.

Table 4 - Post-traumatic growth—quality assessment of studies (quantitative and mixed methods) using Mixed Methods Appraisal Tool (MMAT)
First author (year) Type of study Screening questions Quantitative nonrandomized studies
Are there clear research questions? Do the collected data allow to address the research questions? 3.1. Are the participants representative of the target population? 3.2. Are measurements appropriate regarding both the outcome and intervention (or exposure)? 3.3. Are there complete outcome data? 3.4. Are the confounders accounted for in the design and analysis? Quality
x/****
Castellano-Tejedor et al (2017) [29] Quantitative Yes Yes Yes Yes Yes Yes ****
Findler et al (2014) [17] Quantitative Yes Yes Can't tell Yes Yes Yes ***
Ford et al (2018) [18] Quantitative Yes Yes Yes Yes Yes Yes ****
Gardner et al (2015) [20] Quantitative Yes Yes Can't tell Yes Yes Yes ***
Gardner et al (2017) [19] Quantitative Yes Yes Can't tell Yes Yes Yes ***
Hensler et al+ (2013) [34] Mixed methods Yes Yes Yes Yes Yes No a ***(*)
Kamibeppu et al (2010) [28] Quantitative Yes Yes Yes Yes Yes Yes ****
Schepers et al (2018) [21] Quantitative Yes Yes Yes Yes Yes Yes ****
Turner-Sack et al (2016) [22] Quantitative Yes Yes Can't tell Yes No Yes **
Wataru et al (2019) [23] Quantitative Yes Yes Can't tell Yes Yes Yes ***
Willard et al+ (2016) [35] Mixed methods Yes Yes Can't tell Yes Yes No a **(*)
Yonemoto et al (2012) [15] Quantitative No Yes Yes Yes Yes Yes ****
Zebrack et al (2011) [24] Quantitative Yes Yes Can't tell Yes Yes b Yes ***
NOTE: For qualitative and quantitative studies: * = 1 criterion met; ** = 2 criteria met; *** = 3 criteria met; **** = 4 criteria met. For mixed methods studies, the lower of the 2 is taken as the overall quality score; + = mixed method study.
aSample size likely too small to identify associations.
bSiblings used as comparisons; consequently, the results were not always reported in detail.

2.6. Data synthesis

We narratively synthesized the evidence for reported levels and domains (aim 1 and 2) of PTG and associated sociodemographic and psychosocial characteristics in families of CCS (aim 3).[13] An assimilation approach was applied to combine the results from quantitative and qualitative studies.[14] A meta-analysis of results was not feasible because of statistical heterogeneity of the included articles, inclusion of qualitative studies, and the relatively low number of studies that were retrieved and could be included.

3. Results

3.1. Search results

We retrieved 646 articles on PTG, and one additional study[15] was identified from the reference list of the included studies (Fig. 1).

3.2. Study characteristics

Sixteen articles that met the eligibility criteria for PTG were published between 2010 and 2021. All studies were cross-sectional, including 11 quantitative,[15–29] 3 qualitative,[30–33] and 2 mixed methods.[34–36] One project used the same sample of participants in 2 studies.[19,20] We extracted evidence from all these studies, but participants were only counted as 1 sample per project. Seven studies originated from the United States,[18–21,24,34,35] 3 from Japan,[15,23,28] 2 from Canada,[22,31] and 1 each from Spain,[29] Israel,[17] New Zealand,[32] and Sweden.[30] Seven studies used comparisons.[17,18,21–26,28] Family members were compared with other members of the same family (eg, sibling of CCS with CCS) or compared with other families with either healthy children or children with chronic illness (Table 2).

3.3. Participants of the studies

Seven studies included both parents,[15,19–21,23,29,30] one only mothers,[35] one only fathers,[31,34] and one grandparents.[17] One qualitative study included the closest support person, that is, 6 were mothers, 2 partners of survivors, and 1 grandmother.[31] One study investigated both parents and siblings[37]; 4 studies focused on siblings.[18,24,28,32] The sample size of the studies varied from 9 to 2820. The total number of caregivers was 758 (196 fathers, 503 mothers, 57 grandparents, and 2 other relationships) and 3280 siblings (1756 female, 1524 male).

The children's ages at diagnosis ranged from 0 to 20 years. The mean time since cancer diagnosis was 12.0 years (range: 5.8–23.8years). Mean time since end of treatment was 5.7 years (range: 3.6–8.8 years). Seven studies did not report time since treatment completion.[17,18,23,24,28,30,31] The main characteristics of the included studies are summarized in Table 2.

3.4. Assessment of quality

Overall, the quality of PTG studies was good. Two studies[22,35] met 2/4 criteria, 6[17,19,20,23,24,34] met 3/4 criteria, and 8 studies[15,18,21,28–32] met all (4/4) criteria. Incomplete quality was most often because of lack of reporting whether and which characteristics had been taken into account as potential confounders (Tables 3 and 4).

3.5. Narrative synthesis

In this systematic review, we narratively synthesize the evidence for caregivers (parents including mothers and/or fathers, as well as other support persons, and grandparents) and siblings of CCS. When studies reported about a specific subgroup of caregivers, we referenced it accordingly.

3.5.1. Levels and domains of PTG in caregivers

Twelve studies focused on PTG in caregivers.[15,17,19–23,29–31,34,35] Five studies used the PTGI,[15,17,21–23] and 3 studies used the BFS.[19,20,34] Studies have shown higher PTG in parents[21] and grandparents[17] of CCS than in parents or grandparents of healthy children. One study further showed that parents' PTG was significantly higher than survivors'.[22] However, no difference in levels of PTG was found when parents of CCS were compared with parents of children with chronic disease.[23] Levels of PTG and benefit finding were found average to high, with items often endorsed as either “quite a bit” or “extremely.”[15,22,23,34] Caregivers often reported high “personal growth”[17,30,34,35] and “relating to others,”[30,31,34] whereas the domains of “spiritual change,”[34] “new possibilities,”[31] and “appreciation of life”[30] were only found to be high in caregivers in 1 study each.

Five studies investigated general positive psychological changes.[29–31,34,35] Castellano et al found 59% of parents reported both positive and negative consequences, and an additional 22% reported uniquely positive consequences after their child's cancer experience.[29] Almost all fathers and mothers reported positive consequences, such as personal development, improved relationships, changed faith or shift in perspective, and receiving social support.[30,34,35] In addition, mothers were also found to report child-specific growth as part of their perceived positives consequences, such as their child's character becoming “funnier” or “braver.”[35] Oftentimes, few fathers were part of a study; those who reported on fathers showed similar patterns of PTG and benefit findings as found among mothers.[15,21,29,30,34]

3.5.2. Levels and domains of PTG in siblings

Four studies measured PTG in siblings of CCS using the PTGI,[18,22,24,28] with siblings showing either lower[22,24,28] or no difference[18] in PTG to CCS. By contrast, when comparing siblings with matched controls, siblings of CCS reported higher “appreciation of life” than the healthy control group.[28] Some sex differences were found in “total PTG” and “relating to others,” where female siblings of CCS reported higher PTG than female siblings of matched controls; while these differences were not found for male siblings.[28] One study revealed concerningly low levels of PTG in siblings.[22]

Only one study explored siblings' experiences using a qualitative approach.[32] All siblings (n = 10) reported experiencing some degree of positive psychological change.[32] Most reported “personal growth,” such as siblings perceiving to have become more independent and that reflecting on the cancer experience may have hastened their maturity.[32] A little less than half of siblings reported stronger family relationships, even if their parents had separated.[32] Changes included improved communication, stronger sibling bond, and appreciating family time.[32] A majority of siblings felt more optimism after the cancer experience of their siblings.[32]

3.5.3. Characteristics associated with PTG

3.5.3.1. Sociodemographic.

Seven studies[15,19–22,34,35] investigated sociodemographic characteristics and found no associations between family members of CCS and PTG outcomes.[15,20,22,34,35] Variables studied included parents' age, sex, ethnicity, religion, family composition, rurality, socioeconomic status (SES), level of education, income, and marital status. One study found family income positively associated with benefit finding; however, the significance disappeared after adjusting for psychological variables in the model.[20] Another study by the same group and using the same sample identified sociodemographic characteristics (ie, age, marital status, socioeconomic status, and living environment [rurality]) as a moderator between levels of benefit finding and quality-of-life outcomes.[19]

3.5.3.2. Clinical.

Five studies investigated the clinical characteristics of CCS such as sex, age at diagnosis, diagnosis, length of treatment, time since diagnosis, and potential sequelae.[15,20,22,23,29] No significant associations were found with PTG. Only 1 study found that caregivers of children diagnosed with leukemia and those treated with only 1 modality (eg, surgery or chemotherapy or radiation) reported higher benefit finding, but these associations disappeared after adjusting for psychological characteristics.[20,29]

3.5.3.3. Psychological.

Three studies[15,19,23] reported a relationship between PTSS and PTG and found mixed results: Yonemoto et al[15] found higher levels of PTSS were associated with greater PTG in parents of survivors of osteosarcoma cancer. By contrast, Wataru et al[23] did not find PTSS to be associated with PTG among the parents of CCS. Two further studies examined the relationship between coping and PTG[20,35]: Maternal caregivers of CCS, who could reappraise the childhood cancer experience from a positive perspective, were seen to use emotion-focused and problem-focused coping strategies more frequently, and this was associated with greater benefit finding.[35] Similarly, being more optimistic was associated with greater benefit finding.[20] Caregivers of CCS who used a higher level of active, emotion, or positive spiritual coping strategies were also more likely to report benefit finding.[20] Wataru et al[23] showed that parents of CCS with more re-examination of core beliefs reported greater PTG. There was no significant positive relationship between intrusive rumination and PTG.[23]

3.5.3.4. Social.

Three studies found a positive association between social support or available emotional resources and PTG.[17,20,35] Findler et al found that the more social support grandparents of children with chronic illness received, the more PTG they reported; however, this was not the case in grandparents of CCS.[17] Interestingly, 1 study reported less relational frustration in the parent-child relationship and higher attachment as a result of the cancer experience and compared with parents of healthy children.[21]

3.5.3.5. Siblings.

Only 1 study reported on associations between sociodemographic and clinical characteristics with siblings' PTG outcomes, with no significant associations found.[22]

4. Discussion

The aim of this review was to update the most recent evidence regarding PTG in family members of CCS.[7] A total of 16 relevant articles were analyzed. Although PTG was measured in different ways, overall, the synthesis suggests that a majority of family members experience some form of positive psychological change after the childhood cancer experience.

We have seen that family members of CCS are most likely to show “personal growth” and enhanced “relating to others.”[4] Families were found to experience closer family relations reporting that the cancer experience made them become stronger as a family unit.[28,30,31,34] These conclusions are consistent with Duran's previous review,[7] who found most families to report a positive aftermath for family life, including an increased self-knowledge and closer family relationships after the childhood cancer experience.[7]

Among siblings, we found “personal growth” was reported most often, with feeling more mature and independent after the cancer experience.[32] Previous studies have reported similar outcomes, with siblings of children on active cancer treatment and CCS themselves reporting to mature.[7,9] When comparisons were made with healthy controls, higher PTG was found among parents, grandparents, and siblings of CSS.

Although Tedeschi and Calhoun's framework has suggested that sex may predict PTG,[4] findings have been inconsistent. Based on the current review, we did not find any stark sex differences in family members' PTG.[37] Given that very few studies have identified associations with other sociodemographic characteristics, modifiable psychosocial characteristics may play a bigger role in the development of PTG than more stable biological and social contextual structures.

The evidence presented in this review supports the role of available emotional resources such as social support and proactive coping skills in the development of PTG. Social support was positively associated with PTG in caregivers of CCS and particularly well received if provided by the partner or another family member.[17,20,35] This finding is in line with the PTG framework, and a longitudinal study on patients with cancer demonstrated that receiving support from a spouse or close family members was a promoting characteristic for PTG.[5,38] Similarly, the review of Picoraro et al[39] found that support from close family members was associated with higher PTG among parents of children with pediatric illness. We also found that higher PTSS, higher optimism, and proactive and religious coping were associated with higher PTG in parents of CCS.[15,19,20,23,35]

Taken together, social support and coping strategies in times of adversity seem to promote PTG, and this may be one reason why families of CCS often reported becoming closer as a family. This is important given that characteristics such as family cohesion, being able to plan for the future, and good perceptions of self have been identified as positively associated with better parental mental health.[25]

4.1. Limitations and strengths

All quantitative studies included in the present systematic review were cross-sectional with no longitudinal design; therefore, it remained difficult to determine how PTG evolved over time in families of CCS. Many of the characteristics associated with PTG have been investigated only by a few studies, and studies have applied different methodologies to investigate PTG. Owing to the scarcity of studies in the field regarding PTG, we extended the term “survivor” to include all children who completed cancer treatment independently of the time since end of treatment or since diagnosis. Usually, the term “survivor” is reserved for children who had been diagnosed more than 5 years ago. The levels and domains of PTG experiences may change over time. The search was limited to studies published in English, which may have excluded relevant studies in other languages.

Despite these limitations, this review acknowledges the latest evidence regarding PTG in childhood cancer. Four databases were systematically searched using comprehensive search terms. Searching the reference lists of the included articles allowed confidence that most relevant research was included. Moreover, 2 authors (B.H., J.B.) independently screened the selection of articles, appraised the studies for their methodological integrity (quality assessment), and cross-checked the extraction of data.

4.2. Future directions and clinical implications

Longitudinal, prospective, family-based studies assessing psychosocial measures are needed to gain a more holistic understanding of how families develop and maintain positive psychological changes over time. While sociodemographic characteristics are often hardly modifiable, and were not found to be associated with PTG, we suggest to further investigate and broaden our understanding of modifiable, psychosocial characteristics that may inform clinical practice and family interventions. Psychological characteristics have remained largely understudied—oftentimes these are modifiable and thus are important variables to study to identify potential points for intervention, promoting PTG in families affected by childhood cancer. In particular, the role of coping strategies in families of CCS is poorly understood, and further investigation is warranted to better understand the psychological adjustment to the childhood cancer experience.

Our review showed that social support and proactive coping strategies were associated with increased PTG. Informal social support, such as an individual's existing social network, or formal social support, which can be from professionals or nonprofessionals, such as trained peer group facilitators or members may be a helpful source for families.[40] Healthcare professionals may play a crucial role in communicating to families of CCS about the importance of social support and proactive coping may play a key role in referring them to tailored services during and after treatment.[41,42] The need to focus on modifiable characteristics and further understanding of the ways in which social support and coping drive PTG in family members of CCS may be particularly crucial. People who successfully cope with a difficult event often build confidence and perceive themselves as stronger (eg, increased perceived self-efficacy), which they may apply to future challenging situations.[43]

5. Conclusion

Evidence from the present review shows that characteristics such as psychological well-being, personal resources, and coping strategies may play a bigger role in the development of PTG than sociodemographic characteristics. We found limited research regarding PTG in families of CCS in the past decade, especially regarding siblings, despite a previous review emphasizing the importance of more research to inform clinical practice. While families reported little growth in spiritual change or in finding new possibilities in life, they experienced greater sense of personal growth and stronger family relationships.

Funding

Dr. Julia Baenziger is the recipient of an early postdoctoral mobility fellowship from the Swiss National Science Foundation (SNSF grant P2LUP1_195091).

Authors' contribution

Berglind Halldorsdottir: primary data analysist, main contributor to the manuscript.

Gisela Michel: designed the scope of the study, provided feedback on analysis and manuscript (interpretation of data, writing, revising)

Julia Baenziger: codesigned the scope of the study, secondary analysist, provided feedback on analysis and manuscript (interpretation of data, writing, revising).

Acknowledgement

We wish to thank all authors of the studies which are included in this review for their contribution to this field. This article is based on a revised version of the master thesis “Post-traumatic growth and resilience in families of CCS—A systematic review” that has been submitted to the Department of Health Sciences & Medicine at the University of Lucerne in 2020. The master thesis has been prepared by Berglind Halldorsdottir under the supervision of Gisela Michel and Julia Bänziger. This review did not receive any funding. JB is supported by an early post-doc mobility fellowship Swiss National Science Foundation SNSF grant P2LUP1_195091.

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Keywords:

benefit finding; childhood cancer survivors; coping; families; post-traumatic growth; stress-related growth; systematic review

Copyright © 2022 The Authors. Published by Wolters Kluwer Health Inc., on behalf of the International Psycho-Oncology Society.